Intravenous Immunoglobulin (IVIg) with Methylprednisolone Pulse Therapy for Motor Impairment of Neuralgic Amyotrophy: Clinical Observations in 10 Cases

Naito, Kosuke; Fukushima, Kazuhiro; Suzuki, Shigenori; Kuwahara, Motoi; Morita, Hiroshi; Kusunoki, Susumu; Ikeda, Shu‐ichi

doi:10.2169/internalmedicine.51.7049

Cited by 56 publications

(40 citation statements)

References 21 publications

(30 reference statements)

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“…As it is likely that neuralgic amyotrophy attacks are ultimately induced by an auto-immune response to the peripheral nervous system [1][2][3][4][5], efforts have been undertaken to attempt treatment in the acute phase with immunomodulants such as corticosteroids or iv gammaglobuline [6][7][8][9][10][11]. Unfortunately, a randomized controlled trial of corticosteroids failed because it proved impossible to recruit the needed number of participants within the three-year trial period.…”

Section: Introductionmentioning

confidence: 99%

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

Alfen

Groothuis

Nobacht³

et al. 2015

Neuromuscular Disorders

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ObjectiveNeuralgic amyotrophy is considered a rare peripheral nervous system disorder but in practice seems grossly under recognized, which negatively affects care for these patients. In this study we prospectively counted the one-year incidence rate of classic neuralgic amyotrophy in a primary care setting. MethodsIn a prospective cohort study during the year 2012 we registered all new cases of neck, shoulder or arm complaints from two large primary care centers serving a population of 14,118. Prior to study, general practitioners received a short training on how to diagnose classic neuralgic amyotrophy. Neuralgic amyotrophy was defined according to published criteria irrespective of family history. Only patients with a classic phenotype were counted as definite cases. After inclusion, patients with suspected neuralgic amyotrophy who had not yet seen a neurologist were offered neurologic evaluation for diagnostic confirmation. ResultsOf the 492 patients identified with new onset neck, shoulder or arm complaints, 34 were suspected of having neuralgic amyotrophy. After neurologic evaluation the diagnosis was confirmed in 14 patients. This amounts to a one-year incidence rate for classic neuralgic amyotrophy of 1 per 1000. Data Availability Statement: No centralized data collection was available for this study. Data were collected in an anonymized digital datafile and stored locally by the study group. The primary data file for this study is available as S1 Datafile.Funding: This study was sponsored by an unsolicited and unconditional gift from the Dutch Rare Diseases Fund (Stichting Zeldzame Ziekten Fonds; www.zzf.nl) and the ZABAWAS Foundation (www. zabawas.nl). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. ConclusionsOur findings suggest that neuralgic amyotrophy is 30-50 times more common than previously thought. Unawareness of the disorder and its clinical presentation seems the most likely explanation for this difference. An incidence rate of 1 per 1000 and the long-term sequelae many patients suffer warrant more vigilance in diagnosing the disorder, to pave the way for timely treatment and prevent complications.

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Section: Introductionmentioning

confidence: 99%

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

Alfen

Groothuis

Nobacht³

et al. 2015

Neuromuscular Disorders

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“…The good response of idiopathic brachial plexopathy to immunosuppressive or immunomodulatory treatment has been reported in the literature (1,16).…”

Section: Discussionmentioning

confidence: 95%

“…Electromyography, nerve conduction studies and MRI must be interpreted in light of the patient's clinical history. The correlations between imaging results and clinical and neurophysiological findings may further increase the overall specificity and sensitivity (15,16). Moreover, conducting an imaging workup is useful for ruling out other pathologies that can mimic the symptoms, including primitive and secondary tumours, paraneoplastic immunomediated syndromes, bony and soft tissue thoracic outlet syndromes and joint shoulder disease.…”

Section: Discussionmentioning

confidence: 99%

MRI Neurography Findings in Patients with Idiopathic Brachial Plexopathy: Correlations with Clinical-neurophysiological Data in Eight Consecutive Cases

et al. 2013

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Objective Idiopathic brachial plexopathy is a non-progressive disorder characterized by the sudden onset of shoulder pain associated with weakness and sometimes paraesthesia of the arm. Clinical and electrophysiological examinations are the primary diagnostic tools and allow physicians to localize the site of damage. MRI neurography is rarely performed in this setting. Methods We herein describe the cases of eight consecutive patients suffering from idiopathic brachial plexopathy. All patients underwent clinical visits, neurophysiological evaluations and MRI neurography. Results We confirmed the primary role of clinical and neurophysiological evaluations in the diagnosis of idiopathic brachial plexopathy and demonstrate the usefulness of brachial plexus MRI neurography for confirming the presence of inflammatory changes. Conclusion In patients with idiopathic brachial plexopathy, MR neurography is a helpful tool for excluding different aetiologies, such as compression or tumour formation, and/or confirming inflammatory changes.

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“…The dose regimen described in that study varied between patients, and he proposed that patients take oral PSL at a daily dose of 1 mg/kg for 1 week and gradually stop the medication during the second week [1,11]. Recently, Naito et al reported that intravenous immunoglobulin with methylprednisolone pulse therapy may be a potential treatment for PTS in which the disease is detected 1 month or more after onset [12].…”

Section: Discussionmentioning

confidence: 99%

Parsonage-Turner Syndrome Initially Suspected of Being Orthopedic Diseases in a Primary Care Setting: A Case Report

Yabe

2014

J Med Cases

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Parsonage-Turner syndrome (PTS), also known as neuralgic amyotrophy or brachial neuritis, consists of a complex set of symptoms including sudden onset shoulder pain, usually unilaterally, followed by progressive neurologic deficits such as motor weakness, dysesthesia and numbness. We report a case of a 45-year-old Japanese man with PTS, who was initially suspected of having orthopedic diseases, such as cervical spondylosis and suprascapular nerve entrapment syndrome. The patient presented with acute-onset severe pain in his right arm and neck, followed by muscle weakness. We diagnosed PTS by taking a detailed medical history, ruling out other differential diagnoses, and by considering the characteristic clinical symptoms and the denervation pattern identified by needle electromyography. High-dose corticosteroid therapy prednisolone (PSL) relieved the initial sharp pain and oral amitriptyline successfully relieved the residual nighttime pain. Doses of PSL and amitriptyline were tapered, pain was alleviated, and the patient returned to full-time work after discharge in one-and-a-half years. For prompt diagnosis, physicians should consider the possibility of PTS in patients with acute onset of severe arm pain or subsequent muscle weakness and muscle atrophy in the arm or shoulder girdle.

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Intravenous Immunoglobulin (IVIg) with Methylprednisolone Pulse Therapy for Motor Impairment of Neuralgic Amyotrophy: Clinical Observations in 10 Cases

Cited by 56 publications

References 21 publications

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

Incidence of neuralgic amyotrophy (Parsonage–Turner syndrome) in a primary care setting: A prospective cohort study

MRI Neurography Findings in Patients with Idiopathic Brachial Plexopathy: Correlations with Clinical-neurophysiological Data in Eight Consecutive Cases

Parsonage-Turner Syndrome Initially Suspected of Being Orthopedic Diseases in a Primary Care Setting: A Case Report

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